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Record W2791641983 · doi:10.1093/jcag/gwy009.195

A195 TRIPLE OVERLAP SYNDROME? A RARE CASE OF AIH, PBC AND PSC OVERLAP

2018· article· en· W2791641983 on OpenAlexaff
Waleed Alghamdi, Karim Qumosani

Bibliographic record

VenueJournal of the Canadian Association of Gastroenterology · 2018
Typearticle
Languageen
FieldMedicine
TopicParvovirus B19 Infection Studies
Canadian institutionsLondon Health Sciences CentreWestern University
Fundersnot available
KeywordsMedicineAutoimmune hepatitisUrsodeoxycholic acidOverlap syndromePrimary biliary cirrhosisPrimary sclerosing cholangitisGastroenterologyLiver biopsyInternal medicineBiopsyPathologyHepatitisDisease

Abstract

fetched live from OpenAlex

Autoimmune liver diseases share the principle of immune-mediated damage directed towards either hepatocytes or bile ducts. They are classified into Autoimmune Hepatitis and Primary Biliary Cholangitis (AIH-PBC), AIH and Primary Sclerosing Cholangitis (AIH-PSC) and AIH and undefined cholestatic syndrome. AIH-PBS and AIH-PSC are recognized in the literature. Few case reports presented PBC-PSC overlap. The occurrence of all three entities together is extremely rare. To describe a rare presentation of immune-mediated liver disease with features of AIH, PBC and PSC. Case report and literature review. A 61-year-old Caucasian lady was referred to our hepatology clinic with a diagnosis of PBC for 10 years and subtle features of AIH on liver biopsy. She was on ursodeoxycholic acid (UDCA) dose of 1.5 g/day which was reduced due to weight loss and anorexia. She had no features of cirrhosis. A year after, she experienced severe itching with an increase in her ALT to 150 U/L, AST to 134 U/L and AP to 367 U/L. Her total bilirubin (TBIL) was 12.3 umol/L and direct bilirubin (DBIL) was 6.9 umol/L. Her ANA and SMA levels were negative. Her AMA titre was > 1:320 with IgG level of 17.3, normal IgA and IgM. Her IgG subtypes, p-ANCA and c-ANCA levels were normal along with CA19-9. She, subsequently, underwent a liver biopsy which showed loose granulomas, Batts/Ludwig fibrosis grade of 2 /4 with activity of 1 /4, positive AMA, periportal lymphoplasmacytic infiltrates and patchy interface hepatitis. There was no definite ductopenia. Based on these findings, we increased her UDCA dose back and started her on budesonide 9 mg/day with azathioprine. Her itching along with ALT, AST and AP levels improved with this regimen at 3 months follow up. We then started weaning off her budesonide. Her pancreatic enzymes, TBIL and DBIL increased which led to stopping azathioprine and increasing her budesonide dose. Abdominal imaging showed normal pancreas, splenomegaly with heterogenous liver parenchyma and dilated intrahepatic bile ducts. Gastroscopy showed small esophageal varices and portal hypertensive gastropathy. She remained asymptomatic with no itching. Her TBIL and DBIL levels improved. Her ALT, AST and AP levels were 76, 96, 266 U/L, respectively. MRCP showed moderate intra and extra-hepatic biliary dilatations with multifocal strictures, and extrahepatic duct wall thickening and stenosis with stones and sludge can’t be ruled out. ERCP confirmed MRCP findings and intrahepatic PSC. She is currently maintained on budesonide and UDCA. Her MELD-Na score was 6 at her last clinic visit. To the best of our knowledge, this is only the third reported case presenting with overlapping features of AIH-PBC-PSC together. Treatment of similar rare presentations is usually challenging and individualized with variable responses given the lack of randomized controlled trials (RCTs). None

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.249
Threshold uncertainty score0.808

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.011
GPT teacher head0.257
Teacher spread0.246 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

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Citations4
Published2018
Admission routes1
Has abstractyes

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